RESEARCH ARTICLE Open Access

Functional outcome of surgical treatmentof adults with extremity osteosarcoma aftermegaprosthetic reconstruction—single-center experienceTomasz Goryń*†, Andrzej Pieńkowski†, Bartłomiej Szostakowski, Marcin Zdzienicki, Iwona Ługowska andPiotr Rutkowski

Abstract

Background: Osteosarcoma is the most common primary malignant bone tumor in adults and is usually located inthe long bones. Standard treatment consists of perioperative chemotherapy and radical surgical resection. Limb-sparing surgery using a variety of reconstructive techniques remains the gold standard.

Methods: In our study, we retrospectively analyzed 90 adult patients operated at our institution between 2000 and2017 for extremity osteosarcoma that underwent limb-sparing reconstruction with the megaprosthesis. Sixty-onepatients underwent resection and reconstruction of the distal femur, 9 patients—proximal femur, 7 patients—proximal tibia, 5 patients—total femoral resection and reconstruction, 5 patients—proximal humeral resection, and3 patients—other types of resection with endoprosthetic reconstruction. The median follow-up time was41 months, median overall survival was 86 months (3–225 months), and progression-free survival was 81 months(1–86 months). Functional assessment was made on 48 out of 56 living patients, after endoprostheticreconstruction. The assessment was made according to MSTS functional scale.

Results: In 14 cases (15%), the endoprosthesis had to be explanted, or amputation was performed for localrecurrence or septic complication. Due to a mechanical failure of the implant, we had to perform a revision in 5patients (5%). Eighteen out of 74 patients with endoprosthesis died of the disease. The median MSTS score was84% (53–100%), and the best result of 85% was achieved in patients after distal femoral resection withendoprosthetic reconstruction.

Conclusion: Careful planning of the treatment of patients with extremity osteosarcoma that is performed at thereferral centers gives the possibility of long-term survival with a good and excellent functional result.

Keywords: Osteosarcoma, Combined treatment, Limb reconstruction

BackgroundOsteosarcoma is the most commonly diagnosed primarymalignant bone tumor with bimodal age distribution withits peak attributed to puberty and the fifth decade of life.The incidence of osteosarcoma in population is 0.2 per100.000, which is less than 1% of solid tumors [1, 2].Osteosarcoma commonly occurs in the long bones of the

extremities. The most common sites include two of thebones forming the knee joint, followed by the proximalfemur and the proximal humerus. Due to its rarity,osteosarcoma in adults remains a big challenge fordetermining a proper diagnosis and for setting correcttreatment in a short period of time. The best treatmentresults are obtained with the use of combined therapy thatincludes perioperative chemotherapy, followed by radicalsurgical resection. At present, limb-sparing surgery (LSS)can be performed in the majority of patients withextremity osteosarcoma. Currently, various reconstructive

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* Correspondence: Tomasz.Goryn@coi.pl†Tomasz Goryń and Andrzej Pieńkowski contributed equally to this work.Department of Soft Tissue/Bone Sarcoma and Melanoma, MariaSklodowska-Curie Institute – Oncology Center, Warsaw, Poland

Goryń et al. Journal of Orthopaedic Surgery and Research (2019) 14:346 https://doi.org/10.1186/s13018-019-1379-3

techniques are used to restore the functional limb afterextensive bone resection. The most frequently used areendoprosthetic joint reconstructions, arthrodesis implants,autografts, allografts, and custom-made implants.Despite the increased risk of local recurrence, LSS is

currently considered the optimal treatment solution.Published research shows that patients undergoing suchtreatment are characterized by a better total survival rateas compared with patients after amputation, not to men-tion reducing physical disability and its psychologicaleffects that occur in patients undergoing such mutilatingprocedure [3–5].Amputation is necessary only in 10–30% of patients

with extensive involvement of surrounding soft tissues,vessels, or nerves, in poor general condition or afterimproper, earlier surgical treatment. It is caused mostlyby delay in setting a proper diagnosis and treatment,which leads to more advanced disease at the time ofdiagnosis.Due to more extensive muscle and bone resection in

osteosarcoma patients, their functional results are inferioras compared to patients treated for degenerative disease.Most papers which present the functional outcome of

sarcoma patients after endoprosthetic reconstructioncontain small non-homogenous groups of patients witha variety of diagnoses from metastatic lesions to thewhole spectrum of primary bone tumors.Many of them present the results of patients in a wide

range of ages, from children to older people. In the caseof children, their functional results are usually betterthan adults due to their better adaptation and regener-ation possibility, which helps in more effectiverehabilitation.In most cases, the diagnosis in children is made

earlier, and resection is not as extensive as in adults.The most worldwide adopted/approved scale to meas-ure the functional outcome of patients after limb-sparing surgery is Musculoskeletal Tumor Society(MSTS) scoring scale. This scale can answer the ques-tion of how the patients benefit from a limb-sparingsurgery and allow comparing the variety of recon-struction techniques.

The Department of Soft Tissue/Bone Sarcoma andMelanoma in one of the few referral centers in the Eastand Central Europe dedicated to complex treatment ofthe whole spectrum of bone sarcomas. In our department,patients are treated according to standard proceduresadopted from NCCN and ESMO recommendation.This study aimed to evaluate the functional results of

adult patients treated for limb osteosarcoma in ourreferral center with megaprosthetic reconstruction.

Material and methodsWe had retrospectively analyzed the history of adultpatients treated at our institution due to osteosarcomain years 2000–2017 with the use of inclusion and exclu-sions criteria that are shown on Table 1.Finally, we have qualified 90 patients that underwent

LSS with megaprosthetic reconstruction.The indications for LSS in our group of patients were re-

sectable osteosarcoma without neurovascular involvementand adequate soft tissue envelope for prosthetic coverage.Tumor resectability was assessed on MRI or CT imaging

performed after the third course of preoperative chemo-therapy. According to AJCC classification in patients instage IV of the disease, additionally, all metastases mustbe resectable after the preoperative chemotherapy courses.The resection of metastases was arranged after completingperioperative chemotherapy courses.The chemotherapy was started after pathological

diagnosis, and staging of osteosarcoma was confirmed inopen or needle core biopsy of the tumor.

Table 3 Stage of osteosarcoma according to AJCC classificationv.8 in analyzed group of patients

AJCC Patient number

IA 4

IB 9

IIA 33

IIB 39

III 0

IV 5

Table 1 Study group inclusion and exclusion criteria

Inclusion criteria Exclusion criteria

• Age > 18 years old • Lost of follow-up

• Confirmed osteosarcomain biopsy

• Surgery outside ourdepartment

• Extremity location • Non-extremity location

• Limb-sparing surgery withthe use of megaprothesisin our department

• Mutilating surgery

• Complete follow-up

Table 2 Type of reconstruction, the presence of pathologicalfracture, and the presence of metastases (M1) at the diagnosisin analyzed group of patients

Patient number Distalfemur

Proximalfemur

Femurshaft

Proximaltibia

Proximalhumerus

Others

Resection andreconstruction

61 9 5 7 5 3

M1 4 0 1 0 0 0

Pathologicalfracture

3 3 1 0 1 3

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Routinely, we administered three courses of preopera-tive chemotherapy based on cisplatin and doxorubicin.From 2017 in patients under 30 years of age, we addedmethotrexate to routine chemotherapy. After surgery,patients received between 3 and 6 courses of chemother-apy according to the same preoperative schedule.All patients were operated at our department by an ex-

perienced team of surgeons between 3rd and 4th weeksafter the last course of neoadjuvant chemotherapy.All patients underwent resection of osteosarcoma

followed by reconstruction with megaprothesis.In the first 18 patients treated with endoprosthetic

reconstruction, we used custom-made implants. In therest of patients, we implanted modular endoprosthetictumor replacements (MUTARS by Implantcast GMbHGermany).The minimum follow-up was 12 months from the end

of treatment; the median follow-up time was 41 months.The mean age in the whole group of patients was 34 years(18–68). Patient characteristics are presented in Table 2.In Table 3, the stage of osteosarcoma according to

AJCC staging classification version 8 in the analyzedgroup of patients is shown.All patients were followed-up in our outpatient clinic.

At the time of their appointment, we assessed themagainst the following criteria: pain, function, acceptanceof treatment, support, walking ability, and gait. Eachevaluated category was rated from 0 to 6 points.Functional effect of treatment was assessed in alive

patients with implanted endoprosthesis with the help ofthe MSTS score system: 35 patients (88% alive patients)after distal femur resection and reconstruction, 4 pa-tients (100% alive patients) after proximal femur resec-tion and reconstruction, 2 patients (100% alive) after

total femur resection, 5 patients (100% alive patients)after proximal tibia resection, and 2 patients (100% alive)after proximal humerus resection and reconstruction.

ResultsMedian overall survival (OS) in the whole group ofpatients was 86 months (range 3–225 months), and

Fig. 1 5-year prosthesis survival risk (5-y-PSR)

Table 4 Age, survival time, and stage of osteosarcomaaccording to AJCC classification of dead patients

Patients Age Survival timein months

AJCC stage

1 29 69 IIA

2 21 54 IIA

3 39 52 IIA

4 30 48 IIB

5 59 48 IIB

6 58 47 IV

7 43 43 IIB

8 23 28 IIB

9 19 32 IIB

10 30 22 IIA

11 37 21 IIA

12 20 21 IIB

13 56 21 IIB

14 26 14 IIB

15 30 13 IV

16 34 11 IIB

17 67 9 IV

18 22 3 IV

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progression-free survival (PFS) was 81 months (range 1–86 months).During follow-up, 18 out of 74 patients died from the

disease between 3 and 69 months after the surgery.Age, survival time in months, AJCC stage of patients,

who died, is shown in Table 4.From a group of 90 patients after endoprosthetic re-

construction in 14 cases (15%), the endoprosthesis wasexplanted or the patient was amputated for local recur-rence or septic complication.We removed 9 of 61 (16%) distal femoral endoprosth-

esis, 3 of 5 (60%) proximal humerus, 1 (11%) proximalfemur, and 1 of 7 (14%) proximal tibia endoprosthesis.Five patients (5%) had a revision for mechanical failureof the implant. During follow-up, 18 of 74 patients withendoprosthesis died of the disease.The prosthesis survival curve is presented in Fig. 1.The results of MSTS evaluation are displayed in Figs. 2

and 3.Twenty-four patients (49%) of all assessed patients

achieved MSTS score better than 90% in the whole

group and 20 patients (52%) in the group of patientsafter distal femur resection and reconstruction.Achieved results show that patients had an excellent

functional outcome of treatment, and they could return toa normal life after completing very demanding treatment.The example of the reconstruction and functional re-

sults in a patient with osteosarcoma of the distal femuris shown in Fig. 4.In our series, local recurrence (LR) occurred in 15 pa-

tients (16%), and its presence was related to poorer OS.In a group of patients with LR, metastases occurred in13 patients (86%) and 13 patients (86%) died of thedisease. Comparing to a group of 75 patients without LRwho had much better results, in which metastasesoccurred only in 18 patients (24%) and 14 (18%) of themdied of the disease (Fig. 5).

DiscussionDepartment of Soft Tissue, Bone Sarcoma and Melanomais a Polish referral center for the treatment of adult patientswith the variety of primary bone and soft tissue sarcomas.

Fig. 3 MSTS score results according to category

Fig. 2 Total MSTS score results depending on resection

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The treatment results in this department are similar tothe results obtained in other reference centers.Our treatment protocols are in line with recommended

by ESMO, NCCN, and Polish Society of Oncology.In our group of patients in I–III stages of the disease treated

with resection and reconstruction, the 5-year OS rate is 74%.That results are similar to the results presented by

other European centers [6–9]. Results presented byGerman-Austrian-Swiss group in 1709 patients wereslightly worst (5-year OS rate 49%), but the group alsoincluded patients treated with amputations [10].Local recurrence (LR) occurred in our series in 16

patients (17.7%), and its presence was related to poorer OS.Occurring LR is connected to more aggressive disease anda much worse survival rate. LR is observed much moreoften in patients after non-radical resection (R1). LR oc-curred in 50% of patients with non-radical (R1) resectioncomparing with only 8.5% LR in patients after R0 resection.The main goal of LSS is to avoid disability and to give the

patient a chance to return to normal life after demandingand challenging treatment. In the lower limb, the minimalfunctional effect is to give the patient a chance to mobilizewith support with preserved limb sensation.

In the upper limb, the minimal functional effect ishand grasping ability with preserved sensation. Whenthis minimum requirement cannot be achieved with re-constructive surgery, it should be considered to performamputation with an external prosthetic device. This typeof treatment could give patients a better comfort of lifethan preserving the non-functional limb.Functional results in patients treated at our depart-

ment with megaprosthetic reconstruction can be consid-ered good or very good.The best results were achieved in patients after resec-

tion and reconstruction of tumor located in the distalfemur with an average MSTS score of 85%.More than half of patients from this group achieved

MSTS score of 90% or higher, which means that theyreturned to normal life without disability seen.In the majority of patients, resection and proper recon-

struction allowed them to return to work or even to playrecreational sports. That aspect of treatment is crucial topatients, especially at young age, to whom working or play-ing sport is an essential component of life quality. Theworst functional results were obtained in patients after totalfemoral and proximal tibial reconstruction. These poor re-sults could be caused by a small number of patients whowere evaluated with the tumor in this particular location.The other reason for poor results could be the fact

that resection of a tumor located in the proximal tibia isconnected with the necessity of resection and recon-struction of a patellar ligament in the proximal tibia re-section and resection of muscles stabilizing the hip jointand pelvis in total femoral resection. Above can lead todelayed rehabilitation and irreversible disability.In the majority of patients, reconstruction in the upper

extremity can preserve the hand and elbow function,which helps to maintain an acceptable quality of life.

ConclusionWell planned and performed combined treatment ofadult patients with extremity osteosarcoma leads tolong-term survival with good or excellent functional re-sults in the majority of patients.The correct treatment is the most crucial, non-

disease-related, and modified-able risk factor for obtain-ing patients with long-term survival.Limb-sparing surgery is a gold standard of surgical

treatment, and long-term survival rates are favorable inthat group of patients comparing to a group of patientsafter amputation.Majority of patients treated with resection and megapros-

thetic reconstruction allowed them to return to normal lifeand work. It also reduces physical and mental disability.Obtaining the correct and quick pathological diagnosis

as well as starting the correct treatment in the referral

Fig. 4 Results after resection and reconstruction of 20 cm of theright distal femur

Goryń et al. Journal of Orthopaedic Surgery and Research (2019) 14:346 Page 5 of 7

center with no delay is the key to achieving the best re-sults in patients with osteosarcoma.Establishing accurate and early diagnosis could lead to a

decreasing number of performing amputations and allowedto start treatment in patients with less advanced disease.It is highly recommended to establish the referral cen-

ter network for treating patients with sarcomas, includ-ing bone sarcomas, to give optimal diagnosis andtreatment procedures without delay and to reduce thecost of treatment. That referral units participating ininternational clinical trials allow patients to get the latestand experimental therapies. Our department is a mem-ber of European Reference Network (ERN) for rare adultsolid cancers (EURACAN), which gives our patient thebest treatment opportunities.

Abbreviations5-y-PSR: 5-Years prosthesis survival risk; ERN: European Reference Network;ESMO: European Society for Medical Oncology; EURACAN: EuropeanReference Network for rare adult solid cancers; GMbH: Gesellschaft mitbeschränkter Haftung-private German companies with limited liability;LR: Local recurrence; LSS: Limb-sparing surgery; MSTS: Musculoskeletal TumorSociety; NCCN: National Comprehensive Cancer Network; OS: Overall survival;PFS: Progression-free survival; R1: Non microscopically radical resection

AcknowledgementsWe really appreciate all the nursing staff of the Department of Soft Tissue/Bone Sarcoma and Melanoma Institute - Oncology Center, Warsaw, Poland,for helping to collect and record all data.

Authors’ contributionsTG designed the study, collected and analyzed the data, and drafted themanuscript. AP collected the data and drafted the manuscript. IŁ, PŁR, andMZ analyzed the data. BSZ analyzed and proofread the manuscript. Allauthors read and approved the final manuscript.

FundingThis work was supported by the Institute - Oncology Center, Warsaw, Poland.

Availability of data and materialsThe dataset supporting the conclusions of this article is available onrequest—please contact the corresponding author MD Tomasz Goryń.Administrative permission was received from Maria Sklodowska-Curie Insti-tute - Oncology Center, Warsaw, Poland, to access the medical records.

Ethics approval and consent to participateThe Ethics Committee of Institute - Oncology Center, Warsaw, Poland,approved the study protocol. Written informed consent was obtained fromall patients enrolled in the investigation.

Consent for publicationNot applicable.

Competing interestsThe authors declare that they have no competing interests.

Received: 4 July 2019 Accepted: 13 September 2019

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